Clinical characteristics and effective treatments of scrub typhus-associated hemophagocytic lymphohistiocytosis in children

Jin, Yimei; Liang, Dongshi; Huang, Airong; Zhou, Aihua

doi:10.1016/j.jare.2018.05.007

Cited by 7 publications

(6 citation statements)

References 30 publications

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“…However, some experts disagree with doxycycline as the first choice of treatment, particularly in neonates, because of its potential side effects, and instead prefer azithromycin and clarithromycin ( 24 ). Macrolides, including azithromycin, are considered safe for use during pregnancy and in young children, with a recommended dose of 10 mg/kg/day (maximum 500 mg) for 5 days ( 2 , 25 , 26 ). Furthermore, azithromycin seems to be a favorable option in pregnant women with scrub typhus ( 27 ).…”

Section: Discussionmentioning

confidence: 99%

Congenital scrub typhus: a case report and literature review

Liang,

Su,

Chen

et al. 2023

Front. Pediatr.

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BackgroundThis study aimed to analyze the clinical course of a newborn with congenital scrub typhus caused by vertical transmission and explore early diagnosis and treatment strategies. The clinical data of the neonate were retrospectively analyzed and the related literature was reviewed.Case presentationThe newborn was a full-term one, with a good Apgar score at birth. The neonate had apnea at 3 h of life, requiring NICU care and IV antibiotics (piperacillin) for suspected sepsis. An examination revealed hepatosplenomegaly. Blood tests revealed anemia and thrombocytopenia and a chest x-ray showed patchy inflammation. On the second day of life, he developed a fever. On the third day of life, he required mechanical ventilation because his condition worsened after he presented with dyspnea, hypotension, depressed sensorium, and other signs of sepsis. Importantly, the neonate's mother had a history of scrub typhus at 31+ weeks of gestation. While the blood culture result was still pending, high-throughput sequencing of blood and cerebrospinal fluid was performed. To address the suspected scrub typhus infection, oral azithromycin dry suspension was added to the treatment regimen. High-throughput sequencing results on the 5th day of life confirmed a significant presence of 16SrRNA sequences in the blood, suggesting an Orientia tsutsugamushi infection. The neonate steadily recovered and was discharged 16 days after hospitalization. The neonate was followed up for 9 months, and the outcome was favorable with normal growth and development.ConclusionsThis article reports a case of congenital Orientia tsutsugamushi infection, a rare condition caused by vertical transmission. Our review of the literature, combined with the presented case, brings the total number of documented congenital scrub typhus cases caused by vertical transmission to eight. Regrettably, one patient from this group unexpectedly died on the 10th day of hospitalization, resulting in a mortality rate of 12.5% (1/8). The special transmission mode and clinical manifestations of this disease will serve as an alert to doctors for timely diagnosis and treatment. Because of the non-specific clinical manifestations of congenital scrub typhus, limited understanding, low index of suspicion among clinicians, and a lack of diagnostic facilities, scrub typhus is seriously underdiagnosed in pregnant women, fetuses, and neonates.

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Section: Discussionmentioning

confidence: 99%

Congenital scrub typhus: a case report and literature review

Liang,

Su,

Chen

et al. 2023

Front. Pediatr.

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“…A few reports of rickettsial infection causing hemophagocytic syndrome exist, though this condition occurs more frequently in children with an incomplete immune system ( 17 , 18 ). The patient's condition must be distinguished from disorders such as thrombotic thrombocytopenic purpura (TTP) and hemolytic uremic syndrome (HUS).…”

Section: Discussionmentioning

confidence: 99%

Case report: A case of sepsis caused by rickettsial infection-induced hemophagocytic syndrome

et al. 2023

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Hemophagocytic lymphohistiocytosis (HLH) is a rare histiocytic disorder characterized by reactive hyperplasia of the mononuclear phagocytic system, which is primarily caused by dysfunction of cytotoxic killer cells and natural killer cells, leading to antigen clearance barriers and the overactivation of the mononuclear phagocytic system due to continuous antigen stimulation. HLH encompasses a group of clinical syndromes marked by the overproduction of inflammatory cytokines. A 68-year-old Chinese man presented with persistent fever, chills, nausea, and vomiting; the patient had no history of any underlying conditions. Laboratory investigations revealed decreased levels of red blood cells, white blood cells, and platelets, along with reduced natural killer cell activity, increased CD25, hyperferritinemia, and the detection of Rickettsia DNA in his blood, meeting the diagnostic criteria of the Histiocyte Society HLH-2004 guidelines. The patient was treated with antibiotics, improving anemia, glucocorticoid therapy, and continuous renal replacement therapy (CRRT), temporarily improving his condition. However, the patient died after 2 years from chronic renal failure caused by septic shock.

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“…Masand 21 Ganesh 11 Basu 14 Bal 17 Our study Sample size threatening complication of scrub typhus in children, are grossly deficient and limited to occasional case series. Jin et al 30 reported the beneficial outcome in 15 children with scrub typhus associated HLH with intravenous antibiotics (azithromycin or chloramphenicol) without the need for 2004 HLH protocol chemotherapy. Large sample size, robust back up of data from a single center, and analysis of predictors of severe scrub typhus are the strengths of the study.…”

Section: Discussionmentioning

confidence: 99%

Analysis of 262 Children with Scrub Typhus Infection: A Single-Center Experience

Kispotta

Kasinathan

Kommu

et al. 2021

The American Journal of Tropical Medicine and Hygiene

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Scrub typhus, a vector-borne rickettsiosis, is the leading treatable cause of non-malarial febrile illness in Asia. The myriad of typical and atypical features poses a clinical conundrum. We aimed to study the clinical and laboratory profile of children with scrub typhus infection diagnosed by IgM ELISA. Data of children < 12 years presenting with undifferentiated fever to the pediatric services of a tertiary teaching institute between January 2012 and December 2018 were retrieved. Children with seropositive IgM ELISA (InBios International Kit, Seattle, WA) for scrub typhus were enrolled in the study. Clinical features, laboratory investigations, treatment received, and the outcome recorded were obtained. Objective evidence of organ dysfunction was taken as severe scrub typhus. In total, 262 children were diagnosed with scrub typhus. The mean age was 5 years, with male preponderance (65%). And, 13 children presented during infancy. Fever was universal, and generalized lymphadenopathy (93.5%) and hepatomegaly (70%) were the common clinical signs. Eschar was identified in 31%, with greater predilection for groin and axilla. Thrombocytopenia was striking in one-third of children. Also, 25 children (9.5%) had severe scrub typhus and 18 required intensive care stay. Elevated aspartate aminotransferase enzyme levels was a predictor of severity ([OR 3.9], P value 0.005) by multivariate analysis. Lymphadenopathy was found significantly associated with eschar (P < 0.005). No mortality was recorded. This 6-year study underscores the varied spectrum of pediatric scrub typhus infection. Zero mortality in our cohort signifies the excellent outcome with judicious first-line antibiotics.

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Clinical characteristics and effective treatments of scrub typhus-associated hemophagocytic lymphohistiocytosis in children

Abstract: Graphical abstract

Cited by 7 publications

References 30 publications

Congenital scrub typhus: a case report and literature review

Congenital scrub typhus: a case report and literature review

Case report: A case of sepsis caused by rickettsial infection-induced hemophagocytic syndrome

Analysis of 262 Children with Scrub Typhus Infection: A Single-Center Experience

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